ECE2016 Eposter Presentations Clinical case reports - Thyroid/Others (71 abstracts)
An unusual case of adult onset multi-systemic Langerhans cell histiocytosis with perianal and subsequent incident thyroid involvement
Ozen Oz Gul 1 , Pinar Sisman 1 , Erdem Gozden 2 , Soner Cander 1 , Meral Kurt 3 , Ozlem Saraydaroglu 4 , Turkay Kirdak 5 , Canan Ersoy 1 & Erdinc Erturk 1
1Department of Endocrinology and Metabolism, Uludag University Medical School, Bursa, Turkey; 2Department of Hematology, Uludag University Medical School, Bursa, Turkey; 3Department of Radiation Oncology, Uludag University Medical School, Bursa, Turkey; 4Department of Pathology, Uludag University Medical School, Bursa, Turkey; 5Department of Surgery, Uludag University Medical School, Bursa, Turkey.
Introduction: Langerhans cell histiocytosis (LCH) is a rare sporadic disease characterized by histiocytic neoplastic infiltration of various organ systems and a wide spectrum of clinical manifestations, ranging from benign and self-limiting to lethal.
Case report: We report a rare case of adult onset multi-systemic LCH in a 36-year-old male patient with an initial perianal presentation and incidental finding of subsequent thyroid gland involvement in the follow up period. The patient with history of perianal LCH treated with surgical excision and local radiotherapy was referred to our endocrinology department upon detection of hypermetabolic nodular lesions in the left lateral lobe of thyroid gland on Positron Emission Tomography - Computed Tomography (PET/CT) scan in the 9th month of follow up. Current evaluation revealed euthyroid status, a hypoechoic solid lesion of 13×9 mm in size with irregular borders in the left thyroid lobe on thyroid USG and diagnoses of suspected oncocytic lesion, Hashimoto thyroiditis or LCH based on cytologic assessment of thyroid nodule fine needle aspiration of biopsy. The patient underwent total thyroidectomy and pathological assessment confirmed the diagnosis of Langerhans cell histiocytosis. Assessments in the 6th month of postoperative follow up revealed euthyroid status with no thyroid tissue remnants or pathological lymph node on thyroid USG. In view of the multifocal lesions indicating multi-system disease, a systemic chemotherapy protocol with combination of prednisone and vinblastine has been planned by the hematology department.
Conclusion: Our findings underscore the importance of awareness of this rare condition associated with low index of suspicion among practitioners for LCH as well as the significance of ensuring proper follow-up in adult onset LCH to rule out systemic disease. Along with implementing a treatment plan matching the prognostic stratification of the patient, multidisciplinary approach and long-term close follow up with PET/CT scan in the management of adult onset LCH seem crucial given its rarity and varied presentation.
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Endocrine Abstracts
ISSN 1470-3947 (print) | ISSN 1479-6848 (online)
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